Evidence mapPaperPMID 41518520Full record

ArticleNeurology and therapy2026

Two-Year Outcomes Following Delandistrogene Moxeparvovec Treatment in Ambulatory Patients with Duchenne Muscular Dystrophy: Phase 3 EMBARK Trial.

Jerry R Mendell, Francesco Muntoni, Craig M McDonald, Eugenio M Mercuri, Emma Ciafaloni, Hirofumi Komaki, Carmen Leon-Astudillo, Andrés Nascimento, Crystal Proud, Ulrike Schara-Schmidt and 13 more

Registry-linked trialAbstract read
In one paragraph

Article in Neurology and therapy, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT05096221 (A Phase 3 Multinational, Randomized, Double-Blind, Placebo-Controlled Systemic Gene Delivery Study to Evaluate the Safety and Efficacy of SRP-9001 in Subjects With Duchenne Muscular Dystrophy), which is not on this map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT05096221 phase3completednot on this map

A Phase 3 Multinational, Randomized, Double-Blind, Placebo-Controlled Systemic Gene Delivery Study to Evaluate the Safety and Efficacy of SRP-9001 in Subjects With Duchenne Muscular Dystrophy (EMBARK)

TypeinterventionalSponsorSarepta Therapeutics, Inc.Ran2021 to 2024Enrolled126ConditionsDuchenne Muscular DystrophyArmsdelandistrogene moxeparvovec, placebo
3 · Its place in the literature

Who cites it

3 citing papers in PubMed.

  1. Trial
  2. Article
  3. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

23 authors.

Jerry R MendellCenter for Gene Therapy, Nationwide Children's Hospital, Columbus, OH, USA. JMendell@sarepta.com.ORCID http://orcid.org/0009-0006-3966-6303
Francesco MuntoniDubowitz Neuromuscular Centre, NIHR Great Ormond Street Hospital Biomedical Research Centre, Great Ormond Street Institute of Child Health and Institute of Neurology, University College London, & Great Ormond Street Hospital Trust, London, UK.
Craig M McDonaldUC Davis Health, Sacramento, CA, USA.
Eugenio M MercuriPediatric Neurology Institute, Catholic University and Nemo Pediatrico, Fondazione Policlinico Gemelli IRCCS, Rome, Italy.
Emma CiafaloniUniversity of Rochester Medical Center, Rochester, NY, USA.
Hirofumi KomakiTranslational Medical Center, National Center of Neurology and Psychiatry, Kodaira, Tokyo, Japan.
Carmen Leon-AstudilloDepartment of Pediatrics, University of Florida, Gainesville, FL, USA.
Andrés NascimentoNeuromuscular Unit, Neuropaediatrics Department, Hospital Sant Joan de Déu, Fundacion Sant Joan de Déu, CIBERER - ISC III, Barcelona, Spain.
Crystal ProudChildren's Hospital of the King's Daughters, Norfolk, VA, USA.
Ulrike Schara-SchmidtDepartment of Pediatric Neurology, Center for Neuromuscular Disorders in Children and Adolescents, University Clinic Essen, University of Duisburg-Essen, Essen, Germany.
Aravindhan VeerapandiyanDepartment of Pediatrics, Division of Neurology, University of Arkansas for Medical Sciences, Arkansas Children's Hospital, Little Rock, AR, USA.
Craig M ZaidmanDepartment of Neurology, Washington University in St Louis, St Louis, MO, USA.
Matthew FurgersonSarepta Therapeutics, Inc, Cambridge, MA, USA.
Kai DingSarepta Therapeutics, Inc, Cambridge, MA, USA.
Preeti SinghSarepta Therapeutics, Inc, Cambridge, MA, USA.
Rachael PotterSarepta Therapeutics, Inc, Cambridge, MA, USA.
Damon R AsherSarepta Therapeutics, Inc, Cambridge, MA, USA.
Alexander P MurphyRoche Products Ltd, Welwyn Garden City, UK.
Carol ReidRoche Products Ltd, Welwyn Garden City, UK.
Gregory HooperRoche Products Ltd, Welwyn Garden City, UK.
Carmen O TorreRoche Products Ltd, Welwyn Garden City, UK.
Marianna ManfriniF. Hoffmann-La Roche Ltd, Basel, Switzerland.
Louise R Rodino-KlapacSarepta Therapeutics, Inc, Cambridge, MA, USA.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

introductionDelandistrogene moxeparvovec is a recombinant adeno-associated virus rhesus isolate serotype 74 vector-based gene therapy that addresses the absence of functional dystrophin in Duchenne muscular dystrophy (DMD). EMBARK is a phase 3, two-part, crossover, randomized, placebo-controlled trial assessing the safety and efficacy of delandistrogene moxeparvovec (single intravenous dose 1.33 × 10

methodsAs a result of the crossover study design, 2-year functional outcomes of patients receiving delandistrogene moxeparvovec in part 1 of EMBARK were compared, by pre-specified analysis, with a matched propensity score-weighted external control (EC).

resultsAt 2 years, EMBARK patients showed statistically significant benefit versus the EC cohort in functional outcomes prognostic for delaying loss of ambulation (NSAA, Time to Rise, 10-m Walk/Run), demonstrating sustained stabilization or slowing of disease progression. Delandistrogene moxeparvovec micro-dystrophin expression and sarcolemmal localization were maintained over 64 weeks. No new safety signals were observed between week 52 and week 104. Between baseline and week 104, there were no treatment-related deaths, study discontinuations due to adverse events, or clinically significant complement-mediated adverse events.

conclusionsAt 2 years, stabilization or slowing of DMD disease progression was observed in ambulatory male patients with DMD aged 4 to < 8 years receiving delandistrogene moxeparvovec versus a matched EC cohort. Safety was consistent with EMBARK 1-year data and manageable with appropriate monitoring. CLINICALTRIALS: GOV: NCT05096221.

Indexed as

Delandistrogene moxeparvovecDMDDuchenne muscular dystrophyGene therapyNeuromuscular disorders

Identifiers

PMID41518520
PMCPMC12965945

What Socratic holds

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LicenceCC BY-NC
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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.